Abstract
A 12-year-old girl was referred to the Department of Oral and Maxillofacial Surgery for a large cystic lesion, which was gradually increasing in size. We diagnosed her with intraosseous hemangioma based on the magnetic resonance imaging, computed tomography, and angiography findings and performed surgical curettage.
Three months later, there were no recurrences or cosmetic deformities. However, 3 years later, she revisited the Department of Oral and Maxillofacial Surgery with the complaint of gingival hardness. Recurrent cavernous hemangioma was diagnosed based on the imaging features, and the tumor was removed via the same incision site. The histopathological diagnosis of the specimen was recurrent cavernous hemangioma. Herein, we report a rare case of recurrent cavernous hemangioma in the maxilla that was successfully treated. To the best of our knowledge, this is the first report of a recurrent intraosseous hemangioma of maxilla containing multiple teeth has been completely cured without tooth extraction or surgical resection.
Highlights
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Intraosseous hemangiomas are rare, accounting for only 1% of cases of benign bone tumors.
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Complete excision including a surgical margin of sound tissue is the preferred treatment of intraosseous hemangioma.
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We report a rare case of recurrent intraosseous cavernous hemangioma in the maxilla.
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A recurrent hemangioma has been completely cured without tooth extraction or surgical resection.
1
Introduction
Hemangiomas are benign vascular lesions, mostly arising from soft tissues. Intraosseous hemangiomas are rare, accounting for only 1% of cases of benign bone tumors [ ]. They are mainly located in the vertebral column and calvarium and rarely in the facial bones [ ]. The maxilla is infrequently affected, and cavernous hemangiomas in the maxilla usually involve the teeth and alveolar processes. Herein, we report a case of recurrence of cavernous hemangioma in the right maxilla after its surgical removal. All the teeth were preserved in the treatment of recurrent hemangioma.
2
Case report
A 12-year-old girl presented to the oral and maxillofacial surgery clinic at the Dankook University Dental Hospital with a painless, radiolucent lesion in the right maxilla, with progressive enlargement and hardening. She had no history of systemic diseases or trauma. Plain radiography showed a well-defined radiolucent cystic lesion in the right maxillary sinus that extended from the canine to the first molar and had caused root resorption ( Fig. 1 A). Coronal cone-beam computed tomography (CT) of the lesion involving the teeth and alveolar processes showed significant expansion of the right maxilla with some periosteal bone formation, which suggested a slow rate of expansion of the lesion ( Fig. 1 B). Magnetic resonance imaging (MRI) revealed a well-defined homogenous mass measuring 3× 2.9 × 2.7 cm which was hypointense on T1-weighted MRI and markedly hyperintense on T2-weighted MRI ( Fig. 1 C).
Transfemoral cerebral angiography (TFCA) was performed with the suspicion of vascular malformation ( Fig. 1 D). The main feeding vessel of the tumor was the internal maxillary artery, but embolization was not performed, as there were no enlarged feeding vessels or rapid arteriovenous shunting.
Complete excision involving the surgical margin was required to prevent tumor recurrence. However, considering the age of the patient, we decided to perform curettage to preserve the teeth and facial bone as much as possible.
First, an aspiration of the sinus, attempted under general anesthesia, revealed only a limited amount of hematic material with minimal bleeding. Secondly, the anterior maxillary wall was exposed with the Caldwell-Luc approach. We observed tumors protruding into the anterior maxillary wall and achieved curettage while preserving the stability of the teeth and alveolar processes. The tumors were removed from the maxilla and teeth, and subsequently, the residual bones and roots of the exposed teeth were thoroughly scraped using surgical curettes.
We removed all tumors from the palatal mucosa, and the palatal region was curetted and cauterized, with caution to avoid perforation. There was no bleeding tendency at the time of surgery, and nasal filling was performed to prevent postoperative bleeding. The histopathologic diagnosis was intraosseous cavernous hemangioma of the maxilla ( Fig. 2 ).
